Wilson D B, Wyatt D P
Department of Surgery, School of Medicine, University of California, San Diego, La Jolla 92093-0604.
J Neuropathol Exp Neurol. 1993 May;52(3):253-9. doi: 10.1097/00005072-199305000-00009.
The pathogenesis of lumbosacral spinal dysraphism was analyzed in vitro in early homozygous embryos of the vl (vacuolated lens) mutant mouse, and comparisons were made between the lag in closure of the posterior neuropore at the beginning of culture and the degree of severity in the spinal defect at the end of culture for each embryo. In the majority of abnormal (vl/vl) embryos, the neural tube closed completely in culture, although mild defects were observed in the spinal roofplate comparable to those that occur in vivo. Although some abnormal embryos did exhibit small openings in the caudal neural tube at the end of the culture period, in none of the cultured embryos was the extent and severity of the defect as great as that observed in some abnormal embryos of comparable age obtained in vivo. Moreover, the degree of delay in closure of the posterior neuropore at the outset of culture did not necessarily correlate with the severity of the defect obtained at the end of culture. Thus, the expression of the neural tube defect in this mutant appears to be modulated and attenuated by biochemical and/or mechanical factors which may be peculiar to the culture milieu.
在vl（晶状体空泡化）突变小鼠的早期纯合胚胎中对腰骶部脊柱神经管闭合不全的发病机制进行了体外分析，并比较了每个胚胎在培养开始时后神经孔闭合延迟情况与培养结束时脊柱缺陷的严重程度。在大多数异常（vl/vl）胚胎中，神经管在培养过程中完全闭合，尽管在脊髓顶板中观察到了与体内发生的缺陷类似的轻微缺陷。虽然一些异常胚胎在培养期结束时确实在尾侧神经管出现了小开口，但在任何培养的胚胎中，缺陷的程度和严重程度都不如在体内获得的同龄异常胚胎中观察到的那样严重。此外，培养开始时后神经孔闭合延迟的程度不一定与培养结束时获得的缺陷严重程度相关。因此，该突变体中神经管缺陷的表达似乎受到培养环境特有的生化和/或机械因素的调节和减弱。
